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A Review of Melioidosis Cases Imported Into Europe

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European Journal of Clinical Microbiology & Infectious Diseases (2019) 38:1395–1408 https://doi.org/10.1007/s10096-019-03548-5 REVIEW A review of melioidosis cases imported into Europe Sarah Le Tohic1 & Marc Montana2,3 & Lionel Koch4 & Christophe Curti2,5 & Patrice Vanelle 2,5 Received: 8 February 2019 /Accepted: 25 March 2019 /Published online: 4 April 2019 # Springer-Verlag GmbH Germany, part of Springer Nature 2019 Abstract Melioidosis is a tropical bacterial infection, rarely encountered, and poorly known by clinicians. In non-endemic areas, a misdiagnosis can lead to a fatal outcome. This study aims to identify the main characteristics of imported and diagnosed melioidosis cases in Europe to increase clinician’s awareness of this diagnosis. A literature review of imported and diagnosed human melioidosis cases in Europe was performed. PubMed and Web of Science search engines were used for retrieving articles from 2000 to November 2018. Seventy-seven cases of imported melioidosis into Europe described in the literature were identified. More than half of the cases were acquired in Thailand (53%) by men (73%). Patients were usually exposed to Burkholderia pseudomallei during a holiday stay (58%) of less than 1 month (23%) and were hospitalized during the month following their return to Europe (58%). Among travelers, melioidosis is less often associated with risk factor (16%), diabetes being the most frequently comorbidity related (19%). The clinical presentation was multifaceted, pneumonia being the most common symptom (52%), followed by cardiovascular form (45%) and skin and soft tissues damages (35%). The diagnosis was obtained by culture (92%), often supplemented by morphological, biochemical, and molecular identification (23%). Misdiagnoses were common (21%). Over half of the patients received a complete and adapted treatment (56%). Mortality is lower for returning traveler (6%). Imported melioidosis cases into Europe have their own characteristics. This possibility should be considered in patients with pneumonia, fever, and/or abscess returning from endemic areas even years after. Keywords Melioidosis . Pneumonia . Travel medicine . Case reports Introduction Melioidosis is a tropical bacterial infection, caused by a bacil- Electronic supplementary material The online version of this article lus Burkholderia pseudomallei, formerly known as (https://doi.org/10.1007/s10096-019-03548-5) contains supplementary Pseudomonas pseudomallei, Malleomyces pseudomallei,or material, which is available to authorized users. Bacillus whitmori. Each year, there are nearly 165,000 cases worldwide and 54% are estimated to be fatal [1]. The mortal- * Sarah Le Tohic ity burden is similar to that of measles [2]. Melioidosis is [email protected] endemic in Southeast Asia, Northern Australia, India, and China. This disease sporadically occurs in tropical areas be- 1 ’ Hôpital d Instruction des Armées Laveran, Marseille, France tween the 20th of northern and southern parallels [3]. 2 Laboratoire de Pharmaco-Chimie Radicalaire, Aix Marseille Univ, Melioidosis is extremely rare in the temperate zone: they are CNRS, ICR, Marseille, France, Marseille, France essentially imported cases by travelers or immigrants [4]. But 3 Assistance Publique-Hôpitaux de Marseille (AP-HM), Oncopharma, global warming and mass international travel in Asia can in- Hôpital Nord, Marseille, France crease the incidence in non-endemic areas [2]. 4 Institut de Recherche Biomédicale des Armées (IRBA), Pseudomonas pseudomallei is an environmental Gram- Brétigny-sur-Orge, France negative bacterium found in soil and surface water, especially 5 Service central de la qualité et de l’information pharmaceutiques during the rainy season. Community-acquired infections in (SCQIP), Assistance Publique-Hôpitaux de Marseille (AP-HM), human results from exposure through wounded skin, inhala- Marseille, France tion, or ingestion. Nosocomial infections have been rarely 1396 Eur J Clin Microbiol Infect Dis (2019) 38:1395–1408 reported [5]. Melioidosis is not considered to be a contagious of Science (Thomson Reuters Scientific—https://www. infection and human-to-human transmission is very rare [3, 5]. webofknowledge.com/) to identify all published case reports This emerging disease is pleomorphic and opportunistic of melioidosis imported in humans into Europe. The literature because it can affect various organs and displays different search included English-, German-, and French-language ar- clinical presentations from chronic disease to sepsis and death. ticles and publications in other languages if informative ab- Nevertheless, the leading form of melioidosis is pulmonary stracts in the English language were available. disease, as this pathogenic agent is unknown in non-endemic Search terms were selected to detect case reports published areas and it can be easily mistaken for other diseases, such as about imported melioidosis applying exclusion criteria. The community-acquired pneumonia or tuberculosis. So, it is often following terms combinations were applied: Bmelioidosis^ referred to as Bthe Great Mimicker^ [4]andremains OR BBurkholderia pseudomallei^ OR BPseudomonas underdiagnosed. The first diagnosis is based on biochemical pseudomallei^ OR BMalleomyces pseudomallei^ OR tests and morphology identification of bacterium. Molecular BWithmore bacillus^ in human. The Boolean operators were biology is also essential to limit the risk of confusion but takes varied according to the rules of each specific database. longer to obtain. However, early diagnosis and treatment are Searches were conducted using the final limit dates of crucial to reduce mortality, which may reach up to 87% with- January 1, 2000 and November 15, 2018. out treatment [6]. Currently, no vaccine is available and Pseudomonas pseudomallei is widely resistant to several an- Study selection tibiotics, such as gentamicin, colistin, and rifampicin. The bacterium is most often susceptible to ceftazidime, The review was performed in two main stages, as described by meropenem, and amoxicillin–clavulanate. Matos [11]. Eligibility criteria were predetermined by the au- In most European countries, melioidosis is not a nationally thors (Table 1). In the first step, they independently reviewed notifiable disease, making it difficult to ensure epidemiologi- the titles and abstracts yielded by this comprehensive search cal surveillance. However, Pseudomonas pseudomallei is a and subsequently selected articles based on the predetermined selected agent according to the US federal law and belongs inclusion criteria. Any reservations on eligibility for inclusion to Tier 1 agent list of the Centers for Diseases Control and were resolved by discussion between reviewers. After remov- Prevention (CDC). In France, the possession of this organism al of duplicates, the authors read each selected full text and is subject to the regulation on highly pathogenic microorgan- eliminated articles fitting the exclusion criteria. During this isms and toxins (MOT) and must be handled in a biosafety second stage, the references of relevant articles were exam- level 3 laboratory. All these rules and regulations are ex- ined to identify additional cases not found in computerized plained by the high pathogenicity of the bacteria and by its databases. possible use as bioterrorism agent [7–9]. Melioidosis has the potential to become a real public health Data extraction process problem, considering its rising incidence, misdiagnosis, ther- apeutic difficulties, high mortality, and induced biological risk The data collected from all included studies are as follows: [2, 7, 10]. So, we decided to conduct a systematic review of year and country of diagnosis or publication, basic demo- melioidosis cases imported and diagnosed in human in Europe graphic characteristics (age, gender, ethnicity, and socio- since 2000 to increase clinicians’ awareness of its diagnosis. professional category), underlying diseases, foreign travel his- tory (type, country, and duration (< 1 month, 1–6months,> 6months[4], or unknown)), route of exposure, manifestations Materials and methods and care in endemic area, time between arrival in Europe and hospitalization (< 1 month, 1–6 months, > 6 months [4], or Background definitions unknown), clinical manifestations (included fever, pulmonary symptoms, weightless [12],, abscess and localization, bacter- The search method employed in this systematic review was to emia), source of the B. pseudomallei isolates, method used identify and select case reports of imported melioidosis in (phenotype, molecular biology, spectrometry), complementa- humans in Europe. Information on clinical characteristics, ex- ry examinations, researched pathogen agents and misdiagno- posure history, comorbidities, management, and outcome sis, treatments and duration, observed adverse effects, resis- were abstracted. tance to recommended antibiotics, length of follow-up, and outcome (cure, recurrence, death, and unknown). Data sources and searches Morphological and biochemical identification (VITEK 1 and 2, API 20 NE (bioMérieux, Marcy l’Etoile, France)) are We searched MEDLINE/PubMed (National Library of grouped under the name of the phenotype. Molecular biology Medicine—https://www.ncbi.nlm.nih.gov/pubmed)andWeb includes sequencing of 16SrRNA, PCR (polymerase chain Eur J Clin Microbiol Infect Dis (2019) 38:1395–1408 1397 Table 1 Inclusion and exclusion criteria Parameter Inclusion Exclusion 1 Language German, English, French, other languages if Any other
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